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We present a very rare case of neonatal atlantoaxial rotatory subluxation (AARS) and its management.A term female neonate born via vacuum-assisted delivery presented with distress immediately after birth. On examination, she had right-sided torticollis and an audible cervical spine ‘click’. Imaging confirmed AARS. Spinal precautions were initiated, and she was transferred to a surgical tertiary children’s hospital. A rigid cervical collar was applied initially with limited success for immobilisation. Hence, after a multidisciplinary team meeting, it was changed to a rigid spinal immobiliser. Minor immobiliser modifications were required to address challenges with breastfeeding and skin pressure injuries. Her neurological status remained normal throughout her hospital stay. The immobiliser was weaned from 12 weeks and ceased 16 weeks after the initial subluxation.Neonatal AARS can present immediately after birth, necessitating prompt diagnosis and management. Patient-centric, customised spine immobilisation using a collaborative approach can achieve good outcomes for neonatal AARS.